Obstructive sleep apnea syndrome (OSAS) is a relatively common problem in childhood, having a prevalence of 0.7%

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1 Adenotonsillectomy Improves Sleep, Breathing, and Quality of Life But Not Behavior EVELYN CONSTANTIN, MD, ANDREA KERMACK, BSC, GILLIAN M. NIXON, MD, LEE TIDMARSH, MD, FRANCINE M. DUCHARME, MD, AND ROBERT T. BROUILLETTE, MD Objective To obtain parental perspectives on changes in sleep, breathing, quality of life (QOL), and neurobehavioral measures after adenotonsillectomy. Study design This retrospective cohort study comprised otherwise healthy children evaluated for obstructive sleep apnea syndrome (OSAS) from 1993 to We compared those children who underwent adenotonsillectomy with those children who did not. The parents of 473 children (292 boys) 2 years of age and older were sent questionnaires to evaluate QOL and clinical and behavioral changes. For 94 children 3 years of age and older, behavioral changes were evaluated using the Conners Parent Rating Scale-Revised (CPRS-R) for three different periods: pre-operatively/pre-polysomnography, postoperatively/ postpolysomnography, and recently. Results One hundred and sixty-six questionnaires were returned (35%), 138 of which were complete with written consent provided. Compared with parents of unoperated children, parents of children who had adenotonsillectomy were more likely to report improvements in sleep, breathing, and QOL but not improvements in concentration, school performance, and intellectual or developmental progress. Both short and long term, there were no significant effects of adenotonsillectomy on any of the CPRS-R behavior subscales. Conclusion From a parental perspective, adenotonsillectomy frequently improves sleep, breathing, and QOL but does not often improve neurobehavioral outcomes. (J Pediatr 2007;150:540-6) Obstructive sleep apnea syndrome (OSAS) is a relatively common problem in childhood, having a prevalence of 0.7% to 3%. 1,2 OSAS in children is a disorder of breathing during sleep characterized by upper airway obstruction that disturbs sleep and disrupts normal respiratory gas exchange. 3-5 Adenotonsillar hypertrophy is the most common etiology of OSAS in children. For most children, complete resolution of OSAS is achieved with adenotonsillectomy. When a diagnosis of OSAS is not recognized or remains untreated, serious sequelae may ensue, including cor pulmonale and failure to thrive. 4-8 Over the past 10 years a number of studies have reported adverse behavioral and developmental consequences of OSAS. Several studies have reported a relationship between sleep disordered breathing and neurobehavioral and cognitive deficits A few studies have evaluated neurobehavioral outcomes and quality of life(qol) in the months following adenotonsillectomy. As parents have the opportunity to observe their child s behavior and development over prolonged periods, it is important to understand what parents can tell us about responses to medical and surgical interventions. We hypothesized that, several years after adenotonsillectomy, parents would report improved sleep, breathing, QOL, and neurobehavioral and cognitive deficits in children with OSAS, as compared with children who did not have adenotonsillectomy. Furthermore, we expected that the improvement would be sustained and that the greatest improvements would be in the children with the most severe OSAS. To test these hypotheses, we sent questionnaires to parents of children who were evaluated for possible OSAS with subsequent treatment with adenotonsillectomy (the intervention group) and to parents of those who did not have adenotonsillectomy (the comparison group). ADHD ANOVA CPRS-R 540 Attention deficit hyperactivity disorder Analysis of variance Conners Parent Rating Scale-Revised MOAHI OSAS QOL Mixed obstructive apnea/hypopnea index Obstructive sleep apnea syndrome Quality of life From the Departments of Pediatrics (E.C., A.K., F.M.D., R.T.B.) and Psychiatry (L.T.), Montreal Children s Hospital, McGill University, Montreal, Canada; and the Department of Respiratory and Sleep Medicine, Monash Medical Centre, and the Monash Institute of Medical Research, Monash University, Melbourne, Australia (G.M.N.). Evelyn Constantin was supported by a fellowship from the Fonds de Recherche en Sante du Quebec, the Montreal Children s Hospital Research Institute and the Canadian Child Health Clinician Scientist Program. Gillian Nixon was supported by the Alan Ross fellowship of the Department of Pediatrics at the Montreal Children s Hospital/McGill University. Submitted for publication Apr 13, 2006; last revision received Dec 1, 2006; accepted Jan 19, Reprint requests: Dr Evelyn Constantin, Pediatric Sleep Laboratory, Montreal Children s Hospital, 2300 Tupper Street, C508, Montreal, Quebec H3H 1P3, Canada. evelyn.constantin@muhc.mcgill.ca /$ - see front matter Copyright 2007 Mosby Inc. All rights reserved /j.jpeds

2 METHODS Study Population The study cohort consisted of children between 2 and 17 years of age at the time of polysomnography who were evaluated for possible OSAS between January 1993 and December Subjects with asthma were included, but we excluded children with other significant lung diseases, and those with neuromuscular, cardiac, craniofacial, or genetic disorders. We also excluded children evaluated in the sleep laboratory for reasons other than diagnosis of OSAS (ie, neuromuscular weakness, Continuous Positive Airway Pressure (CPAP) titration, or central hypoventilation). Our Institutional Review Board granted ethical approval for the study. Questionnaires and informed consent forms were sent to each child s parent or legal guardian in the spring of We sent reminders and follow-up questionnaires to those parents who had not returned the questionnaires. We did not include in our study any questionnaire responses that were returned after June 1, We collected the following for each patient: (1) cardiorespiratory data from nocturnal polysomnography; (2) pre-polysomnography parental questionnaire 8 ; and (3) follow-up parental questionnaire designed for the current study. Polysomnography Overnight polysomnography was conducted either in our sleep laboratory or at the subject s home using our home polysomnography montage Our home polysomnography system, which includes calibrated respiratory inductive plethysmography, pulse oximetry, and audiovisual recordings, has been shown to give results equivalent to those from laboratory polysomnography for mixed/obstructive apnea/hypopnea index, desaturation index, and respiratory arousal index. Sleep efficiency and the total sleep time were slightly higher in the home compared with the laboratory. 29 Laboratory polysomnography followed the 1996 American Thoracic Society standards. 3 Scoring of polysomnography was performed manually by 30-second epochs. 32 An obstructive apnea was defined as a reduction in the sum signal on calibrated respiratory inductance plethysmography to 20% of baseline for 3 seconds, paired with paradoxical movements of the ribcage and abdomen. Obstructive hypopnea was scored as a reduction in the sum signal to 20% to 50% of baseline for 3 seconds, associated with a drop in SaO 2 of 4%. The desaturation index was defined as the number of drops in SaO 2 of 4% per hour. The respiratory arousal index was defined as the number of respiratory-related arousals per hour of total sleep time. The mixed obstructive apnea/hypopnea index (MOAHI), calculated as the number of mixed obstructive apneas and hypopneas per hour of total sleep time, was used to define OSAS when there was at least one event per hour. 33,34 Pre-polysomnography Questionnaire At the time of the initial clinical referral to the Sleep Laboratory, each parent or guardian had completed a validated sleep laboratory questionnaire. 8 In this first questionnaire, parents were asked to provide demographic data and information about their child s past medical and surgical history, as well as information regarding snoring frequency and loudness, difficulties breathing awake or asleep, and excessive daytime sleepiness. Parents were also asked to report any problems with behavior and/or development. Based on information about snoring, difficulty breathing during sleep, and apneas, an OSA score was calculated. 8 Follow-up Questionnaire (OSAS Outcomes and Neurobehavioral Assessment) The questionnaire mailed to the parents consisted of two parts: one focusing on OSAS outcomes and the other addressing neurobehavioral outcomes, using the Conners Parent Rating Scale-Revised (CPRS-R). 35,36 SLEEP, BREATHING, AND QUALITY OF LIFE OUTCOMES. In the first part of the follow-up questionnaire, parents provided demographic details and information about what treatment, if any, their child received following the initial assessment. Because our primary hypothesis concerned outcomes after treatment, we divided our subjects into two groups: those who had undergone adenotonsillectomy and those who had not. We asked before-after questions to assess for changes in the months following adenotonsillectomy, or following the polysomnography if no surgery had been performed. The questions covered several dimensions: QOL, daytime and sleep breathing, loudness of snoring, asthma, bedwetting, excessive daytime sleepiness, and neurobehavioral/cognitive functioning. The parental responses were based on a 4-point scale: improved, no change, worsened, and not sure. We recoded the latter three responses into one group ( not improved ), for comparison with the improved group. We asked parents to inform us if their child has been diagnosed with attention deficit hyperactivity disorder (ADHD) by a physician and, if so, if the child was on any treatment for ADHD (medication, behavioral treatment, psychological treatment, or no treatment). NEUROBEHAVIORAL OUTCOMES. For children 3 years of age or older at the time of initial polysomnography, we asked parents to complete a questionnaire based on the CPRS-R, a widely used, validated measure of child behavior for children 3 to 17 years of age. Twenty-seven questions were used to evaluate behavior in three time periods: (1) In the months before sleep study/surgery ; (2) In the months after sleep study/surgery ; and (3) In the past few months. The 27 questions were reported by parents/guardians using a 4-point scale ( not true at all, just a little true, pretty much true, and very much true ). The CPRS-R permits evaluation of four subscales: Oppositional, Cognitive/Inattention, Hyperactivity, and ADHD index. The scores were tabulated and Adenotonsillectomy Improves Sleep, Breathing, and Quality of Life But Not Behavior 5412

3 converted to age- and sex-adjusted standardized t scores. 36 All calculations were conducted in duplicate by two different co-authors (EC and AK), who were blinded to the rest of the patient information and questionnaire results. T scores have an average of 50, a standard deviation of 10, and a range of 0 to 100. A t score 60 for each subscale has been reported to be correlated with a diagnosis of ADHD. 10,20 A difference of 10 points (1 standard deviation) in the t score has been suggested as a clinically significant change. 37 The parent-reported short-term and long-term CPRS-R scores for the each of the four subscales were compared with the baseline scores, using repeated-measure analysis of variance (ANOVA). Statistical Analysis We compared the No adenotonsillectomy group with the Adenotonsillectomy group. We analyzed our nominal data using 2 and Fisher s exact tests; continuous data were analyzed using unpaired t tests and Wilcoxon s rank sum tests, as indicated. Our primary hypotheses were that adenotonsillectomy would improve sleep, breathing, QOL, and neurobehavioral and cognitive deficits in the months after adenotonsillectomy, and that the improvement would be sustained. The 10 OSA outcomes measured in the follow-up questionnaire were analyzed using binary backward logistic regression for improvement or non-improvement. The primary intervention of interest was adenotonsillectomy or not; other covariates for the analyses included OSAS or not, MOAHI as a continuous variable (to assess for severity of OSAS), age, and sex. To evaluate whether parents recognized neurobehavioral improvement in children after adenotonsillectomy, analyses were conducted for each of the CPRS-R subscales, to compare changes in two time periods compared with baseline: In the months after sleep study/surgery and In the past few months. The four CPRS-R subscales provided continuous data allowing analysis using three-way repeated measures ANOVA; adenotonsillectomy and a polysomnographic diagnosis of OSAS were independent factors and time period was the repeated measure. A P value.05 was considered to be statistically significant for all analyses, with the exception of the logistic regression analysis, where we applied the Bonferroni correction for multiple comparisons and considered a P value of.005 as statistically significant. The Statistical Package for the Social Sciences (SPSS), version 12 for Windows (SPSS Inc., Chicago, Ill) was used for data analysis. RESULTS Description of the Cohort The recruitment pathway is schematically shown in Figure 1 (available at No difference was found between responders and the nonresponders for age at time of polysomnography, sex, OSA score, MOAHI, and mean saturation or minimum saturation during sleep. Complete data were available for 138 (31%) eligible subjects for the first part of the questionnaire on sleep, breathing, and QOL outcomes; 94 of 109 subjects who were 3 years of age or older had complete data for the neurobehavioral outcome (CPRS-R) section of the questionnaire (Figure 1). Of the 138 subjects, 75 had a diagnosis of OSAS, and 63 did not meet the criteria for OSAS. Adenotonsillectomy was performed on 87% (65/75) of subjects with OSAS and 33% (21/63) of subjects without OSAS. As this was a retrospective cohort study, we have no information on the reasons why the parents and otolaryngologists decided to go forward with adenotonsillectomy in children without polysomnographydocumented OSAS or why they decided not to operate on children with polysomnography-documented OSAS. The cohort included a wide range of sleep-related airway obstruction from normal to severe OSAS (MOAHI range, 0 to 55.1). The age and sex distribution of the subjects evaluated for possible OSAS were characteristic of those usually reported for children with OSAS secondary to adenotonsillar hypertrophy (Table I). 3,33 The adenotonsillectomy group was younger than the group that did not have surgery, but it had similar sex distribution. As expected, the adenotonsillectomy group had a higher OSA score and more severe OSAS as determined by polysomnography (Table I). Sleep, Breathing and Quality of Life Outcomes Compared with children who had not undergone adenotonsillectomy, parents of the adenotonsillectomy group more frequently reported improvement in breathing, snoring, excessive daytime sleepiness, and QOL postoperatively (Table II). Parental reporting of asthma, bedwetting, concentration, school performance, and intellectual or developmental progress were not statistically different between the two groups. Including age and sex as covariates in the logistic regression analysis did not affect these findings. Surprisingly, sleep, breathing, and QOL also improved following adenotonsillectomy in children who did not have OSAS at baseline (n 21). In addition, severity of OSAS, using MOAHI as a continuous variable in the logistic regression model, did not influence the likelihood of improvement after adenotonsillectomy on any medical or neurobehavioral measure. We conducted post hoc analyses to evaluate the effect of early versus late parental recall for the OSA outcomes. The mean time between questionnaire date and surgery date (or questionnaire and polysomnography date, if no surgery was done) was approximately 3.5 years. We chose this midpoint of 3.5 years to designate recall time as early ( 3.5 years) versus late ( 3.5 years). Using the Fisher s Exact Test, there was no statistically significant effect on all OSA outcomes (with the exception of bedwetting) for early versus late recall groups. Neurobehavioral Outcomes Table III shows the four Conners Parent Rating Scale- Revised subscale scores at baseline, short-term follow-up, and 5422 Constantin et al The Journal of Pediatrics May 2007

4 Table I. Descriptive data for the total study group and the two subgroups (no adenotonsillectomy and adenotonsillectomy groups) Study group (n 138) No adenotonsillectomy Group (n 52) Adenotonsillectomy Group (n 86) P value Female (n (%)) 65 (47.1) 26 (50.0) 39 (45.3).72 Age in years at time of PSG 5.5 (3.0) 7.4 (3.5) 4.6 (2.2).01 Age in years at time of T&A N/A N/A 5.0 (2.2) N/A Age in years at time of follow-up questionnaire response 9.6 (3.8) 10.9 (4.2) 8.8 (3.2).001 OSA score 1.3 (2.5) 0.2 (2.6) 1.9 (2.3).001 Total sleep time 8.3 (1.3) 8.1 (1.3) 8.4 (1.4).22 Sleep efficiency (%) 90.1 (7.7) 91.4 (6.1) 89.4 (8.4).12 MOAHI (#/h) 5.5 (9.7) 1.5 (3.7) 7.9 (11.3).001 Respiratory Arousal Index (#/h) 3.0 (5.0) 1.1 (2.1) 4.2 (5.8).001 Mean SaO 2 (%) 97.6 (1.3) 97.8 (1.3) 97.5 (1.4).18 Minimum SaO 2 (%) 88.0 (8.3) 91.5 (4.0) 86.0 (9.4).001 Desaturation Index (#/h) 4.3 (7.6) 2.0 (4.8) 5.6 (8.6).002 Values are expressed as mean (standard deviation). P values determined comparing no adenotonsillectomy and adenotonsillectomy groups. MOAHI, mixed/obstructive apnea/hypopnea index; PSG, polysomnography; T&A, adenotonsillectomy. Table II. Percentage of subjects improving by parental report No adenotonsillectomy group Adenotonsillectomy group P value OR* CI* Quality of life , 71.8 Daytime breathing , 39.9 Sleep breathing , Loudness of snoring , 83.9 Excessive daytime sleepiness , 9.6 Asthma , 7.6 Bedwetting , 15.2 Concentration , 6.5 School performance , 3.6 Intellectual or developmental progress , 2.6 CI, confidence interval; OR, odds ratio. *Adjusted for age and gender. Table III. Conners Parent Rating Scale-Revised (CPRS-R) subscales* CPRS-R subscales No adenotonsillectomy group (n 40) Adenotonsillectomy group (n 54) Baseline Short-term Long-term Baseline Short-term Long-term Oppositional Cognitive/Inattention Hyperactivity ADHD Index Values expressed as mean standard deviation. *Short-term and long-term CPRS-R scores for the each of the four subscales were compared with baseline scores. There were no significant differences found in the four subscales across time, treatment group (the no adenotonsillectomy and the adenotonsillectomy groups), or apnea status (OSA, no OSA). long-term follow-up by adenotonsillectomy status. At baseline, the scores were similar for children with and without OSAS (data not shown) and for those who subsequently had or did not have adenotonsillectomy. Figure 2 (available at shows the shortterm changes in one of the Conners subscales, the Hyperactivity Index, by adenotonsillectomy status; only two subjects had a decrease of at least 10 points after treatment. Other subscale analyses were similar in showing very few subjects improved with or without adenotonsillectomy. Figure 3 shows the long-term changes in the Hyperactivity Index by adenotonsillectomy status; a total of eight subjects had a Adenotonsillectomy Improves Sleep, Breathing, and Quality of Life But Not Behavior 5432

5 Figure 3. Long-term changes in the CPRS-R Hyperactivity Index from baseline to the past few months are plotted against baseline Hyperactivity Index. A Hyperactivity Index 60 (vertical dashed line) is considered to be consistent with a diagnosis of hyperactivity or ADHD. The horizontal dashed line at zero indicates no change in the Hyperactivity Index over time. Note that the majority of children had minimal difference in the Hyperactivity Index, irrespective of whether or not they had surgery. Four children in each group had a decrease of 10 or more. decrease of at least 10 points: four subjects who had adenotonsillectomy and four subjects who had not had adenotonsillectomy. Other subscale analyses were similar in showing that over the long-term, only a few patients in both groups (the Adenotonsillectomy and No Adenotonsillectomy groups) had decreases in the CPRS-R score of 10. There were no statistically significant changes from baseline to both shortterm and long-term changes within any of the four CPRS-R subscales in either the adenotonsillectomy group or the unoperated group (Table III). As for the OSA outcomes, we also conducted post hoc analyses evaluating the effect of early versus late parental recall for the CPRS-R subscales. We performed two-way repeated measures ANOVA with recall time (early versus late) as an independent factor and time period (baseline, short-term, and long-term) as a repeated measure. There was no statistically significant effect on the outcomes of the four CPRS-R subscales for early versus late recall groups. DISCUSSION In this retrospective cohort study, parents of children who had adenotonsillectomy for OSAS frequently described improvements in sleep, breathing, and QOL but rarely reported improvements in behavior as assessed by a validated instrument, the CPRS-R. Notably, the severity of OSAS did not influence the likelihood of improvement after adenotonsillectomy on any of these outcomes, and even children who did not have OSAS on polysomnographic recording showed improvements in sleep, breathing, and QOL, by parental report. A number of studies have reported an association between OSAS and neurobehavioral and cognitive deficits Several groups have found improved neurobehavioral outcomes in children after adenotonsillectomy Ali and his group evaluated 33 children before and after adenotonsillectomy (12 with sleep disordered breathing, 11 snorers, 10 controls). Their study concluded that following surgery, relief of mild to moderate sleep-disordered breathing improves behavior and functioning. 20 Chervin et al and Goldstein et al conducted prospective before-after studies evaluating children s neurobehavioral outcomes following adenotonsillectomy. 21,23 Parent rating scales showed improvement of behavior, 21 inattention, hyperactivity, ADHD, and excessive daytime sleepiness. 23 By contrast to these studies, we found that several years after surgery, parents did not report that their children had improved neurobehavioral outcomes following adenotonsillectomy, either in the period immediately following surgery or in the long term. Moreover, our study included a relatively large sample size of children referred to a pediatric sleep laboratory for evaluation of OSAS; clinical and polysomnographic data were available for all study patients and the follow-up period averaged years. It is possible that recall, observer, information, or selection biases could explain the discrepancy between our results and those of previous investigators. For this retrospective cohort study, the long follow-up can be seen as a disadvantage as well as an advantage. We cannot exclude the possibility that parents had difficulty remembering changes in their child s behavior and development after adenotonsillectomy but had no such difficulty recalling significant improvements in sleep, breathing, and QOL at the same time. However, our analyses revealed no effect of duration of follow-up on OSA outcomes (with the exception of bedwetting) and no effect on outcomes for the CPRS-R subscales. Most previous studies that have evaluated neurobehavioral outcomes after adenotonsillectomy have used psychometric tests measuring such domains as attention and executive function. It may be that parents overlook small changes in such specific domains (observer bias), instead focusing on global development and behavior. Alternatively, in many children, the improvements in cognitive/behavioral functioning after adenotonsillectomy may be too small to be noticed by parents, in the face of all the other impacts on a child s cognition and behavior over time. Numerous tests exist to measure behavior in children. We chose the CPRS-R as our measurement tool for neurobehavioral outcomes because it is a validated, brief, comprehensive and age- and sex-specific measure. However, some of the discordance could be due to test-to-test differences (information bias). More studies are needed to determine the best tests to evaluate neurobehavioral outcomes in children when assessing the impact of interventions to improve sleep quality. Although our study has a relatively large sample size in 5442 Constantin et al The Journal of Pediatrics May 2007

6 comparison with most other studies of behavior in OSAS, our response rate was lower than optimal. To explain our results by selection bias would require the parents of children who had neurobehavioral improvement after adenotonsillectomy to selectively choose not to respond to our follow-up questionnaire. Responders and nonresponders were similar in demographic profiles and polysomnographic metrics, and such a scenario seems unlikely. It is interesting that several children had marked neurobehavioral improvement after evaluation for OSAS. However, these children included both those who had adenotonsillectomy and those who did not. Thus, it remains possible that adenotonsillectomy results in dramatic neurobehavioral improvement in a small percentage of children treated for OSAS, and/or that subgroups, such as children with concurrent ADHD 10 or severe learning problems, 16 may improve. In our study, only 18 of 94 children had a CPRS-R baseline Hyperactivity Index 60, 11 of whom had adenotonsillectomy. Only seven of these 11 children had an improvement in the CPRS-R Hyperactivity Index (four of 7 having a change in their index of more than 10 units). Thus, it will be important both to study such subgroups but also to avoid the temptation to generalize results from such studies to the general population of children with OSAS. Of interest, parents reported improvements of sleep, breathing, and QOL in children who had adenotonsillectomy but who did not have OSAS by polysomnography. Several investigators have found a relation between snoring and behavioral and neurodevelopmental outcomes. 17,38,39 It is possible that polysomnography does not detect all children who will benefit from adenotonsillectomy. 40 One possible explanation is that those children who had a normal polysomnogram but improved after adenotonsillectomy had upper airway resistance syndrome. However, this possibility seems unlikely because our monitoring included calibrated respiratory inductive plethysmography with audiovisual observation, and because our definition of OSAS included patients with a MOAHI as low as one event per hour. Children who were not diagnosed with OSAS rarely showed any increased work of breathing, paradox, snoring-associated arousals, or other evidence of difficulty in breathing during sleep. 29 Several studies have now shown that sleep, breathing, and QOL are likely to improve following adenotonsillectomy for children with OSAS To the extent that parents memories are accurate and that our questions measure the appropriate outcomes, adenotonsillectomy frequently improved sleep, breathing, and QOL but not neurobehavioral outcomes. Thus, pediatricians and otolaryngologists should advise parents that adenotonsillectomy most likely results in these clinical improvements, but physicians should be guarded in promising improvement in behavior and development. We would like to thank the following collaborators for all their hard work and dedication to this project: Sebastien Dube, MSc (Clinical Research Coordinator, Montreal Children s Hospital/ McGill University Research Institute), for his much-appreciated guidance and assistance in our statistical analysis; Vanessa Bonneau and Dawn Creighton, for their enthusiasm and assistance in data collection and database entry; and our sleep laboratory technicians: Angela Morielli, Lois Earle, Melodee Mograss, Sylvia Ladan, Severina Luciano, Jacinthe Lavergne, and Christine McGregor, for obtaining initial questionnaire data and for performing and analyzing the polysomnography data. REFERENCES 1. Ali NJ, Pitson DJ, Stradling JR. Snoring, sleep disturbance, and behaviour in 4-5 year olds. Arch Dis Child 1993;68: Gislason T, Benediktsdottir B. Snoring, apneic episodes, and nocturnal hypoxemia among children 6 months to 6 years old. An epidemiologic study of lower limit of prevalence. Chest 1995;107: Standards and indications for cardiopulmonary sleep studies in children. American Thoracic Society. Am J Respir Crit Care Med 1996;153: Brouillette RT, Fernbach SK, Hunt CE. Obstructive sleep apnea in infants and children. J Pediatr 1982;100: Guilleminault C, Korobkin R, Winkle R. A review of 50 children with obstructive sleep apnea syndrome. Lung 1981;159: Gaultier C. Clinical and therapeutic aspects of obstructive sleep apnea syndrome in infants and children. Sleep 1992;15(6 Suppl):S36-S Menashe V, Farrehi F, Miller M. Hypoventilation and cor pulmonale due to chronic upper airway obstruction. J Pediatr 1965;67: Brouilette R, Hanson D, David R, Klemka L, Szatkowski A, Fernbach S, et al. A diagnostic approach to suspected obstructive sleep apnea in children. J Pediatr 1984; 105: Guilleminault C, Winkle R, Korobkin R, Simmons B. Children and nocturnal snoring: evaluation of the effects of sleep related respiratory resistive load and daytime functioning. Eur J Pediatr 1982;139: Chervin RD, Archbold KH, Dillon JE, Panahi P, Pituch KJ, Dahl RE, et al. Inattention, hyperactivity, and symptoms of sleep-disordered breathing. Pediatrics 2002;109: Chervin RD, Dillon JE, Bassetti C, Ganoczy DA, Pituch KJ. Symptoms of sleep disorders, inattention, and hyperactivity in children. Sleep 1997;20: Chervin RD, Archbold KH. Hyperactivity and polysomnographic findings in children evaluated for sleep-disordered breathing. Sleep 2001;24: Ali NJ, Pitson D, Stradling JR. Natural history of snoring and related behaviour problems between the ages of 4 and 7 years. Arch Dis Child 1994;71: Blunden S, Lushington K, Kennedy D, Martin J, Dawson D. Behavior and neurocognitive performance in children aged 5-10 years who snore compared to controls. J Clin Exp Neuropsychol 2000;22: Owens J, Opipari L, Nobile C, Spirito A. Sleep and daytime behavior in children with obstructive sleep apnea and behavioral sleep disorders. Pediatrics 1998;102: Gozal D. Sleep-disordered breathing and school performance in children. Pediatrics 1998;102(3 Pt 1): Gozal D, Pope DW Jr. Snoring during early childhood and academic performance at ages thirteen to fourteen years. Pediatrics 2001;107: Gottlieb DJ, Chase C, Vezina RM, Heeren TC, Corwin MJ, Auerbach SH, et al. Sleep-disordered breathing symptoms are associated with poorer cognitive function in 5-year-old children. J Pediatr 2004;145: LeBourgeois MK, Avis K, Mixon M, Olmi J, Harsh J. Snoring, sleep quality, and sleepiness across attention-deficit/hyperactivity disorder subtypes. Sleep ;27: Ali NJ, Pitson D, Stradling JR. Sleep disordered breathing: effects of adenotonsillectomy on behaviour and psychological functioning. Eur J Pediatr 1996;155: Goldstein NA, Post JC, Rosenfeld RM, Campbell TF. Impact of tonsillectomy and adenoidectomy on child behavior. Arch Otolaryngol Head Neck Surg 2000;126: Friedman BC, Hendeles-Amitai A, Kozminsky E, Leiberman A, Friger M, Tarasiuk A, et al. Adenotonsillectomy improves neurocognitive function in children with obstructive sleep apnea syndrome. Sleep 2003;26: Chervin RD, Ruzicka DL, Giordani BJ, Weatherly RA, Dillon JE, Hodges EK, et al. Sleep-disordered breathing, behavior and cognition in children before and after adenotonsillectomy. Pediatrics 2006;117:e769-e Flanary VA. Long-term effect of adenotonsillectomy on quality of life in pediatric patients. Laryngoscope 2003;113: De Serres LM, Derkay C, Sie K, Biavati M, Jones J, Tunkel D, et al. Impact of adenotonsillectomy on quality of life in children with obstructive sleep disorders. Arch Otolaryngol Head Neck Surg 2002;128: Goldstein NA, Fatima M, Campbell TF, Rosenfeld RM. Child behavior and quality of life before and after tonsillectomy and adenoidectomy. Arch Otolaryngol Head Neck Surg 2002;128: Mitchell RB, Kelly J, Call E, Yao N. Quality of life after adenotonsillectomy for obstructive sleep apnea in children. Arch Otolaryngol Head Neck Surg 2004;130: Adenotonsillectomy Improves Sleep, Breathing, and Quality of Life But Not Behavior 5452

7 28. Mitchell RB, Kelly J, Call E, Yao N. Long-term changes in quality of life after surgery for pediatric obstructive sleep apnea. Arch Otolaryngol Head Neck Surg 2004;130: Jacob SV, Morielli A, Mograss MA, Ducharme FM, Schloss MD, Brouillette RT. Home testing for pediatric obstructive sleep apnea syndrome secondary to adenotonsillar hypertrophy. Pediatr Pulmonol 1995;20: Morielli A, Ladan S, Ducharme FM, Brouillette RT. Can sleep and wakefulness be distinguished in children by cardiorespiratory and videotape recordings? Chest 1996;109: Mograss MA, Ducharme FM, Brouillette RT. Movement/arousals. Description, classification, and relationship to sleep apnea in children. Am J Respir Crit Care Med 1994;150(6 Pt 1): Rechtschaffen A, Kales A. A manual of standardized terminology, techniques, and scoring system for sleep stages of human subjects. Los Angeles: UCLA Brain Information Service, Brain Research Institute; Marcus CL, Omlin KJ, Basinki DJ, Bailey SL, Rachal AB, Von Pechmann WS, et al. Normal polysomnographic values for children and adolescents. Am Rev Respir Dis 1992;146(5 Pt 1): Uliel S, Tauman R, Greenfeld M, Sivan Y. Normal polysomnographic respiratory values in children and adolescents. Chest 2004;125: Conners CK. A teacher rating scale for use in drug studies with children. Am J Psychiatry 1969;126: Conners CK, Barkley RA. Rating scales and checklists for child psychopharmacology. Psychopharmacol Bull 1985;21: Conners CK. Conners Rating Scales: Revised Technical Manual. North Tonawanda, NY: Multi-Health Systems, Chervin RD, Ruzicka DL, Archbold KH, Dillon JE. Snoring predicts hyperactivity four years later. Sleep 2005;28: Schlaud M, Urschitz MS, Urschitz-Duprat PM, Poets CF. The German study on sleep-disordered breathing in primary school children: epidemiological approach, representativeness of study sample, and preliminary screening results. Paediatr Perinat Epidemiol 2004;18: Goldstein NA, Pugazhendhi V, Rao SM, Weedon J, Campbell TF, Goldman AC, et al. Clinical assessment of pediatric obstructive sleep apnea. Pediatrics 2004; 114: Constantin et al The Journal of Pediatrics May 2007

8 Figure 1. Recruitment pathway. See text for description. Figure 2. Short-term changes in the months after adenotonsillectomy or polysomnography in the Conners Parent Rating Scale-Revised (CPRS-R) Hyperactivity Index are plotted against baseline Hyperactivity Index. Data for 94 subjects 3 years of age are shown. Note that only two subjects had a decrease of 10 or more after adenotonsillectomy; a change in the CPRS-R score of 10 is usually considered significant (improvement or worsening). Adenotonsillectomy Improves Sleep, Breathing, and Quality of Life But Not Behavior 546.e1